Most patients with elbow stiffness have contractures proportional in severity to the initial insult. However, there is a subset of individuals who have stiffness after relatively minor events and who experience repeated early recurrences after soft-tissue contracture release. The purpose of the present report was to describe the clinical features and outcomes for patients presenting with such a condition, which we term refractory elbow arthrofibrosis.
The elbow has a certain propensity for the development of stiffness after injuries or surgery1,2. Contracture of the capsule with extensive fibrosis is a central pathologic element in most stiff elbows, but the severity of capsular fibrosis varies considerably from patient to patient. There is some association between the severity of the initial insult to the joint and the intensity of the fibrotic capsular response. Several studies have documented predictable and permanent improvements in motion when the abnormal capsule is surgically removed3-6.
Over the last few years, we evaluated and managed a subset of individuals who had a severe capsular contracture that was out of proportion to the relatively benign nature of the initial insult and who had a persistent tendency to experience recurrent contractures despite repeated treatment attempts. We are unaware of other reports of this condition. We used strict criteria to include patients in this report, recognizing that there is a spectrum of severity of this condition.
Although the intimate biologic mechanisms underlying this fibrotic response are yet to be elucidated, the purposes of this study were to describe the clinical features and treatment outcome of this condition, to caution against multiple surgical attempts in this patient population, and to stimulate the exploration of alternative diagnostic and potential biologic treatment modalities. Institutional review board approval was obtained. The patients were informed that data concerning their cases would be submitted for publication, and they consented.
Between 1980 and 2003, the records and radiographs of 116 patients referred to the senior author (B.F.M.) for the treatment of extrinsic elbow stiffness (no or minimal contribution of the joint surface to the loss of motion) were reviewed to identify the patients who fulfilled the following inclusion criteria: (1) stiffness following a minor insult to the joint, (2) absence of a known inflammatory or metabolic condition, (3) normal radiographic appearance of the elbow, and (4) failure to improve elbow motion despite several therapeutic interventions. Patients who required excision of heterotopic ossification were excluded from this series. Patients with intrinsic stiffness or those referred to other elbow surgeons at our institution were also excluded.
Four patients fulfilled all four criteria and form the basis of this report. Institutional review board approval was obtained. The history, physical examination, treatment attempts, and final outcome of these patients were reviewed in detail to characterize this pathologic entity. By reported history, all had a normal elbow with no pain or stiffness before the initial insult to the joint. They had been followed at our institution for an average of six years (range, one to ten years).
Patients
The patients included one man and three women with a mean age at the time of presentation to our institution of thirty-six years (range, nineteen to forty-seven years). The initial insult to the elbow included a hyperextension injury, a cortisone injection for lateral epicondylitis, a lateral elbow wound sustained in a motor-vehicle accident, and placement of a peripheral intravenous catheter in one patient each. Before referral to our institution, all patients had had between one and three treatment attempts to improve motion, including two arthroscopic releases (Case 1), splinting followed by open release and manipulation under anesthesia (Case 2), and dynamic splints (Cases 3 and 4).
At the time of presentation, all of these patients complained of mild to severe pain and a severe contracture (Table I). The average ranges of elbow flexion and extension were 81° and 55°, respectively. There were no signs of instability, and the neurovascular examinations were unremarkable. Specifically, these patients had no pain and a negative Tinel sign over the ulnar nerve. They did not present any physical findings consistent with complex regional pain syndrome (reflex sympathetic dystrophy). In addition, they did not share any other additional features that could have made treatment failure more likely, such as mood disorders, lack of employment, or secondary gain, such as a pending Workers' Compensation claim.
Radiographic evaluation both at the time of presentation and throughout follow-up was unremarkable; disuse osteopenia could be appreciated in one patient (Case 1). A bone scan was obtained preoperatively for all four patients and showed moderate increased uptake at the elbow region in all of them. Bone scans are not routinely obtained at our institution prior to contracture release; thus, we do not have data recording bone scan findings for the remaining 112 patients. We obtain a bone scan when there is a high index of suspicion for refractory elbow arthrofibrosis in patients with normal radiographs, as the scan may help to diagnose this condition. These patients were also evaluated for several inflammatory conditions (rheumatoid arthritis, lupus erythematosus, scleroderma, and psoriasis) and metabolic disorders (diabetes mellitus, thyroid disorders, parathyroid disorders, gout, and vitamin-D disorders), and all of these evaluations were unremarkable.
Treatment
The patients encountered at the beginning of the authors' practice were treated more aggressively with repeated treatment attempts with limited success. As the refractory nature of this condition has been recognized, our more recent inclination has been to explain to these patients the nature of the disorder and to be more cautious about repeated treatment attempts.
The four patients included in this report underwent our usual surgical treatment protocol for extrinsic elbow stiffness, which includes anterior and posterior capsulectomies associated with deepening of the olecranon, coronoid, and radial head fossae when needed. In all four patients, the anterior and posterior elbow capsules were thickened and contracted, but the articular cartilage was well preserved.
Postoperative management included continuous passive motion followed by static splints. Recurrence of stiffness was identified within the first few weeks after each procedure and was managed with additional surgery or manipulation with the patient under anesthesia, depending on the interest of each patient to pursue further treatment. These four patients underwent an average of four treatment attempts (range, two to six treatments).
Outcome
The mean amounts of flexion and extension at the first evaluation were 81° and 55°, respectively (Table I). At the most recent follow-up evaluation, all patients continued to complain of moderate pain and had severe stiffness (mean, 83° of flexion and 38° of extension). Motion was lost within the first two months after treatment despite the achievement of a functional range of motion at the time of surgery or manipulation under anesthesia in all four patients. No progressive radiographic change was observed during follow-up.
Refractory elbow arthrofibrosis should be suspected in patients presenting with unexplained elbow stiffness or stiffness after a relatively minor event. Other causes of elbow stiffness can be excluded with radiographs and evaluations for metabolic and inflammatory conditions. Bone scans will show increased uptake at the elbow region. In this subset of patients, treatment should be offered with caution and multiple surgical procedures should probably be avoided.
It could be argued that these four patients did not have a specific condition, compared with the other 112 patients treated for extrinsic elbow stiffness, and that they simply represented the worst four outcomes obtained in a large series of patients. However, we believe these patients behaved with a completely different biologic response after both the initial insult and after treatment attempts, and the recurrences were difficult to explain merely as treatment failures. The frequency of this condition is uncommon as we identified only four patients over a twenty-three-year period.
The intimate biologic mechanisms of elbow contracture are not fully understood. Hildebrand et al. reported increased numbers of myofibroblasts and increased expression of the myofibroblast marker, alpha-smooth muscle actin, in the anterior capsule of six patients treated surgically for elbow stiffness7. In a separate study involving eleven patients, these same authors reported a high rate of capsule matrix turnover with increased mRNA expression for collagen types I, III, and V; biglycan; and matrix metalloproteinases; tissue inhibitors of metalloproteinases were decreased8. Similar findings were reported by Cohen et al.9. These changes are also found in other fibrotic conditions, such as Dupuytren disease and plantar fibromatosis. Refractory elbow arthrofibrosis may represent the intense capsular fibrosis resembling these well-known conditions. Clinically, this condition resembles adhesive capsulitis of the shoulder except the pain is less severe and the elbow contracture does not seem to resolve spontaneously.
Most but not all patients with extrinsic elbow stiffness have improvement in motion after resection of the contracted capsule. Mansat and Morrey reported an average improvement of 45° of motion in thirty-eight elbows treated operatively; however, the arc of motion obtained at the time of surgery was lost at least partially in 26% of the elbows and, at the time of the latest follow-up, 11% of the elbows had not had improved motion5. Other reports have identified patients with recurrent stiffness after elbow contracture release10-12. The patients included in the present study probably represent the more adverse end of the spectrum of recurrence following surgical treatment.
This study has several limitations. It is a retrospective review, and no histopathologic studies were performed on the tissues removed at the time of surgery.
In conclusion, refractory elbow arthrofibrosis is an uncommon response to a relatively minor injury that does not respond to current treatment protocols designed to treat elbow stiffness. Awareness of this condition may help to avoid multiple failed surgical procedures and to stimulate studies to further understand pathologic mechanism(s) and develop alternative treatment strategies. 